Cyproterone acetate and meningioma: a nationwide-wide population based study

Champeaux-Depond, Charles; Weller, Joconde; Froelich, Sébastien; Sartor, Agnès

doi:10.1007/s11060-020-03672-9

Cited by 33 publications

(28 citation statements)

References 35 publications

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“…The suspicion of a link between CPA and meningiomaswas raised by our group in 2008[6]and was con rmedby numerous case seriesshowing regression or stabilization of meningioma(s) after CPA discontinuation [7,8,12,13,15,17,18,27,[29][30][31][32]. Gill et al [14]described an increased risk of meningioma(s)in patients under high dose CPA and with a longer than 1 year exposure.…”

Section: Discussionmentioning

confidence: 99%

“…Since 2009, following the results of our CPA studies [7,8,12,13,20,27], every patient presenting with a meningioma to our neurosurgical department was questioned about previous history of progestin treatment.Every newly diagnosed patient with intracranial tumor radiologically compatible with a meningioma associated with CPA intake, followed aconservative strategy starting with discontinuation of CPA and a close clinical and radiological follow-up.Surgery was onlyindicated in case of tumor growthon serial magnetic resonance imaging (MRI)or in severely symptomatic patients.…”

Section: Patients' Selectionmentioning

confidence: 99%

“…Cyproterone acetate (CPA) is a synthetic progestin drug with strong antiandrogenic effect used for a variety of indications, mostly related to hyperandrogenization and hirsutism in women, while it is used for prostate cancer and paraphilia in men.Thesuspicion of arelationship between the use of CPA and the risk of intracranial meningiomasand, the regression after CPA discontinuation was rst reported in 2008 by Froelich et al[6] and was followed by numerous case reports or series [7][8][9][10][11][12][13][14][15][16][17][18].This increased risk and a strong dose-effect wererecently proven in a large epidemiological study in France [19]. For progestinrelated meningiomas and particularly in the case of CPA, our experience and those of others showed that, when the treatment is discontinued, most of the tumors stabilize in size or decrease, avoiding the need for further surgery [7,8,11,12,15,16,20].…”

Section: Introductionmentioning

confidence: 99%

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Atypical evolution of meningiomatosis after discontinuation of cyproterone acetate: clinical cases and histomolecular characterization

et al. 2021

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Long-term use of cyproterone acetate (CPA) is associated with an increased risk of developing an intracranial meningioma. Discontinuation of CPA most often induce stabilization or regression of the tumor. The exact mechanism of regression is unknown as well as the reason why some meningiomas are still growing after CPA discontinuation.We are reporting four patients with multiple meningiomas, showing opposite tumor evolutions after stopping the CPA highlighting the underlying histologic and genetic features. MethodsPatients presenting several meningiomaswith opposite evolutions following the discontinuation of CPA were identi ed. The clinical and radiological data's were reviewed. A retrospective volumetric analysis of the meningiomas was performed. All the growing meningiomas were operated. Each tumor was characterized histopathologically and by molecular and genetic analyses. ResultsFour female withmultiple meningiomas and opposite tumor volume evolution after CPA discontinuation were identi ed. The histopathological results found broblastic meningiomas for tumorsgrowinglocated in the convexity and a more brousmeningioma in the skull-base tumor which decreased. Meningothelial and transitional meningiomaswere found in two skull-base growing meningiomas.The molecular characterization found twoNF2-mutations among the 4 growing meningiomas. In one patient who presented both patterns, the shrinking skull-basetumor harbored a PIK3CA-mutation whereas the growing tumor, a NF2-mutation. ConclusionTo our knowledge, this is the rst report of such an atypical tumor evolution of CPA-associated meningiomatosis after CPA discontinuation in the same patient. Underlying biological mechanisms explaining this observationespecially, the close relationship between mutational landscapes and the meningeal embryologyin CPA-related meningiomasrequire further research.

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Section: Discussionmentioning

confidence: 99%

Section: Patients' Selectionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Atypical evolution of meningiomatosis after discontinuation of cyproterone acetate: clinical cases and histomolecular characterization

et al. 2021

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“…question of whether the effect of CPA is specific to meningiomata in this region. 29,30 Anatomical location was extracted within this systematic review, but was not reported in all cases; among the 29 reported, 14 (48.3%) were found at the skull base, slightly higher than the expected proportion (*40%). 31 In the wider meningioma literature, the recurrence of grade 1 meningiomas is 7-25% and grade 2 is 29-52%.…”

Section: Findings From This Systematic Review Of Case Reportsmentioning

confidence: 96%

Development and Growth of Intracranial Meningiomas in Transgender Women Taking Cyproterone Acetate as Gender-Affirming Progestogen Therapy: A Systematic Review

et al. 2022

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Background: Gender-affirming hormone therapy is critical to the management of transgender persons. Cyproterone acetate (CPA) is a synthetic, progesterone-like compound commonly used in high doses as gender-affirming progestogen therapy in transgender women. An association between high-dose CPA and the development and growth of intracranial meningioma, including case reports in transgender women, has been described. This systematic review summarizes these cases at the patient level and discusses their management. Methods: This systematic review was registered with PROSPERO (CRD42020191965). A detailed search of the PubMed, EMBASE, and Web of Science electronic bibliographic databases was performed (inception-December 20, 2020). Two review authors independently completed screening, data extraction, and risk of bias assessment in duplicate. Results: Nine records were included describing (n = 12) individual case reports and (n = 35) intracranial meningiomas. The median age at presentation was 48 years (interquartile range [IQR], 43-55 years), most frequent daily CPA doses were 50 mg/day (n = 5) and 100 mg/day (n = 5), and the median duration of CPA use was 9.5 years (IQR, 6.5-17.5 years). Multiple meningiomas were common (n = 7). For most cases (n = 10), surgical resection was the initial preferred management strategy, but two were successfully managed by CPA cessation. Conclusions: Transgender women receiving high doses of CPA may be at increased risk of intracranial meningioma development and/or growth, although this remains a rare disease. For presumed CPA-associated meningioma, drug cessation appears to be an appropriate management strategy when surgery is not imminently required to manage raised intracranial pressure or prevent neurological deterioration. Given the importance of gender-affirming hormone therapy to transgender persons, a suitable alternative hormone regimen should be offered, although the use of CPA in both high doses and for prolonged periods of time is now in decline.

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“…This feature and a higher rate of multiple meningiomas suggest a strong relationship between this hormonal treatment and meningioma development. Therefore, delayed treatment of hormonal induced meningiomas with close clinical and MRI follow-up is recommended by several authors [3,8,9,13,19,22,23,26,28] in asymptomatic patients. Nevertheless, this management still needs further evaluation.…”

Section: Introductionmentioning

confidence: 99%

Have Progestin Related and Sporadic High Grade Meningiomas the Same Natural History?

Devalckeneer¹,

Aboukaïs²,

Faisant³

et al. 2021

Preprint

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Introduction: High grade progestin related meningiomas have been reported in recent series but we found no previous study describing their long-term outcome. Our study aimed to evaluate patients operated on for high grade intracranial meningioma and who underwent long term exposure to high dose of cyproterone acetate, nomegestrol acetate and chlormadinone acetate.Methods: Our study retrospectively included 9 patients with high grade progestin related intracranial meningioma between December 2006 and December 2020. In each patient, clinico-radiological follow-up was performed every 6 months after diagnosis and treatment withdrawal recommendation. Results: The mean progestative exposure was 11.4 years. Edema existence or absence of cleft sign on MRI were the key factors for surgical indication. All patients underwent surgery. Ajduvant radiotherapy was indicated in 1 patient, and Gamma Knife Radiosurgery was proposed in 2 other patients for a second location of mengioma. 6 patients harbored a grade II chordoïd meningioma subtype with 100% PR expression and 3 patients a grade II atypical meningioma subtype with lower PR expression. The mean follow-up was 7.1 years and none of the 9 patients presented with a recurrence.Conclusion: Patients with Grade II progestin related meningiomas have less tumor recurrence after surgery than patients with sporadic high grade meningomas, especially after progestin withdrawal. The presence/ appearance of peri-meningioma edema and the absence of cleft sign before volumetric change should suggest the existence of an underlying high grade meningioma. In these cases, surgical resection may immediately be considered and adjuvant radiotherapy should be reserved for proven recurrence cases.

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Cyproterone acetate and meningioma: a nationwide-wide population based study

Cited by 33 publications

References 35 publications

Atypical evolution of meningiomatosis after discontinuation of cyproterone acetate: clinical cases and histomolecular characterization

Atypical evolution of meningiomatosis after discontinuation of cyproterone acetate: clinical cases and histomolecular characterization

Development and Growth of Intracranial Meningiomas in Transgender Women Taking Cyproterone Acetate as Gender-Affirming Progestogen Therapy: A Systematic Review

Have Progestin Related and Sporadic High Grade Meningiomas the Same Natural History?

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